ALS-related FUS mutations alter axon growth in motoneurons and affect HuD/ELAVL4 and FMRP activity.

doi:10.1038/s42003-021-02538-8

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Article

ALS-related FUS mutations alter axon growth in motoneurons and affect HuD/ELAVL4 and FMRP activity.

Communications biology - 1 Sept 2021

Garone Maria Giovanna, Birsa Nicol, Rosito Maria, Salaris Federico, Mochi Michela, de Turris Valeria, Nair Remya R, Cunningham Thomas J, Fisher Elizabeth M C, Morlando Mariangela, Fratta Pietro, Rosa Alessandro

Abstract excerpt

Mutations in the RNA-binding protein (RBP) FUS have been genetically associated with the motoneuron disease amyotrophic lateral sclerosis (ALS). Using both human induced pluripotent stem cells and mouse models, we found that FUS-ALS causative mutations affect the activity of two relevant RBPs with important roles in neuronal RNA metabolism: HuD/ELAVL4 and FMRP. Mechanistically, mutant FUS leads to upregulation of...

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PMID: 34471224
DOI: 10.1038/s42003-021-02538-8
PMCID: PMC8410767

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